Hindawi

Case Reports in Dentistry

Volume 2018, Article ID 9169208, 3 pages
https://doi.org/10.1155/2018/9169208

Case Report
Cheilitis Glandularis of Both Lips: Successful Treatment with
a Combination of an Intralesional Steroid Injection and
Tacrolimus Ointment

Norberto Sugaya and Dante Migliari

Division of Oral Medicine Clinic, Department of Stomatology, School of Dentistry, University of Sao Paulo, Sao Paulo, SP, Brazil

Correspondence should be addressed to Norberto Sugaya; nnsugaya@usp.br

Received 24 November 2017; Revised 4 February 2018; Accepted 1 March 2018; Published 18 March 2018

Academic Editor: Noam Yarom

Copyright © 2018 Norberto Sugaya and Dante Migliari. This is an open access article distributed under the Creative Commons
Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is
properly cited.

Cheilitis glandularis (CG) is an inflammatory condition of unknown cause that predominantly affects the minor salivary glands of
the lips. Although a diagnosis of CG is not difficult, its treatment is a challenge. This article highlights the clinical presentation of
the disease together with a case of successful management of this disease using a combination of a steroid injection followed by
a topical immunosuppressor.

1. Introduction 2. Case Report

Cheilitis glandularis (CG) is a rare inflammatory condition
that predominantly affects the minor salivary glands and
surrounding tissues of the lips. It affects adults (over 40 years
old) to a greater extent than young people and almost ex-
clusively white individuals. The ratio of male/gender in-
volvement is 3 : 1. To date, no specific factor or cause has been
associated with the disease onset [1].
The clinical features of CG lend to its diagnosis. The
labial mucosa exhibits dilation of the orifices of the minor
salivary glands through which thick saliva (mucin-rich)
flows more intensely due to the inflammatory process in-
side the glandular parenchyma. This excessive salivary flow
eventually dries out, leading to the development of yellowish
plaques (or crusts) that cover the labial mucosa surface.
These plaques are easily removed but form again, mainly
during sleep. In addition to this main feature, the patient also
develops enlargement and eversion of the lips. The vermilion
border is typically not affected [2].
Although a diagnosis of CG is not difficult, its treatment
is a challenge. The present article reports the successful
management of a case treated with a combination of steroid
injection followed by a topical immunosuppressor.
A 16-year-old white male was referred to our oral medicine
clinic for investigation of an enlargement of the lower and
upper lips ongoing for approximately one year. His primary
complaint was yellowish crusts on the mucosa surface of both
lips but particularly the lower one (Figures 1(a) and 1(b)).
Despite removal, the crusts reappeared mostly in the morning.
He underwent previous treatments with topical corticosteroids
to no avail. The patient was healthy with no history of sub-
stantive medical treatment.
Based on the aspect of the lesions, the diagnosis was
cheilitis glandularis. The initial treatment was two intrale-
sional injections of 10 mg triamcinolone suspension in both
lips with a one-month interval between the applications. Two
months following the second injection, an improvement was
noticed with a reduction in the enlargement and eversion of
the lips. Recurrent appearance of the crusts was also reduced
but not to the extent that the patient felt comfortable. Instead
of administering another steroid injection, a topical immu-
nosuppressor (0.1% tacrolimus ointment) was applied twice
daily for two weeks based on two effective management re-
ports on CG found in the literature [3, 4] Before application,
the patient was instructed to (1) wash his hands, (2) remove
2 Case Reports in Dentistry

(a) (b)

(c) (d)

Figure 1: (a, b) Initial consultation with the patient exhibiting yellowish crusts on the labial mucosa. The lips (mainly the lower one) are also
swollen and everted, whereas the vermilion border appears unaffected. (c, d) After treatment, both lips regained normal contour and aspect.
A discreet redness of the lip mucosae remained unchanged throughout the follow-up.

the labial crusts and apply 2% chlorhexidine gluconate on lip
surfaces for disinfection, (3) allow the mucosa surfaces to dry
and then apply the tacrolimus ointment, and (4) wash his
hands again. This management procedure succeeded in
completely resolving the lesions with no recurrence after
a one-year follow-up (Figures 1(c) and 1(d)).
3. Discussion
CG is a rare disease but may occasionally be observed by
the clinician. The lesion appears mainly through a process
of renewed yellowish-plaque formation that is puzzling to
health professionals. It seems that topical corticosteroids
alone are not effective. Intralesional steroid injection has
received some acceptance, but there is no consensus that
it works in all cases. The mainstream treatment for CG
has been vermilionectomy, but this treatment pro-
duces collateral effects, such as permanent itching and
paresthesia.
Our patient did not require a biopsy. Biopsy was judged
unnecessary because there was no clear-cut evidence that
a biopsy would constructively help diagnose CG. Based on
two relevant studies [1, 2], the histopathological findings
were nonspecific, consisting mainly of chronic inflammation
with various degrees of nonspecific sialadenitis and ductal
ectasia of the minor salivary glands and fibrosis within the
glands.
Possible differential diagnoses of CG may include
contact cheilitis and cheilitis granulomatosa [1, 2, 5]. The
first condition is attributed to irritants or allergic con-
tactants. In these cases, apart from disclosing the contactant
agent, the clinical features of contact cheilitis differ sub-
stantially from those of CG. For example, in the former, the
lesions appear more prominent on the lip vermilion and are
characterized mostly by the presence of adherent scales.
Cheilitis granulomatosa shares some similarities to CG
concerning lip swelling and eversion but lacks inflammation
in the labial salivary glands, which leads to an increase of the
salivary secretion and subsequent crust formation, a feature
commonly observed in CG cases.
In the event of a difficulty in making a differential
diagnosis between CG and cheilitis granulomatosa, a bi-
opsy can be helpful as the latter can exhibit noncaseating
granulomas; however, this feature is only observed in 40 to
50% of the cases. In either disease, the exact etiopatho-
genesis remains unknown. In a few cases of cheilitis
granulomatosa, the development of this disease is poten-
tially associated with food allergy (mainly cinnamon and
benzoate compounds) [6].
Although one case of CG is not sufficient to judge the
performance of a therapy, the treatment used in the present
case appears to be effective. Tacrolimus was instrumental in
preventing crust formation, possibly due to its effective
action in controlling glandular inflammation by curbing the
Case Reports in Dentistry 3

production of proinflammatory cytokines inside the minor

salivary glands.

Conflicts of Interest
The authors declare that they have no conflicts of interest.

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